{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T07:19:07Z","timestamp":1790752747978,"version":"4.1.0"},"reference-count":13,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2017,2,23]],"date-time":"2017-02-23T00:00:00Z","timestamp":1487808000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100002426","name":"Fondazione Telethon","doi-asserted-by":"publisher","award":["GGP15098"],"award-info":[{"award-number":["GGP15098"]}],"id":[{"id":"10.13039\/501100002426","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Journal of Sleep Research"],"published-print":{"date-parts":[[2017,8]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>A recently discovered neurodevelopmental disorder caused by the mutation of the cyclin\u2010dependent kinase\u2010like 5 gene (<jats:styled-content style=\"fixed-case\">CDKL<\/jats:styled-content>5) entails complex autistic\u2010like behaviours similar to Rett syndrome, but its impact upon physiological functions remains largely unexplored. Sleep\u2010disordered breathing is common and potentially life\u2010threatening in patients with Rett syndrome; however, evidence is limited in children with <jats:styled-content style=\"fixed-case\">CDKL<\/jats:styled-content>5 disorder, and is lacking altogether in adults. The aim of this study was to test whether the breathing pattern during sleep differs between adult Cdkl5 knockout (Cdkl5\u2010<jats:styled-content style=\"fixed-case\">KO<\/jats:styled-content>) and wild\u2010type (<jats:styled-content style=\"fixed-case\">WT<\/jats:styled-content>) mice. Using whole\u2010body plethysmography, sleep and breathing were recorded non\u2010invasively for 8\u00a0h during the light period. Sleep apneas occurred more frequently in Cdkl5\u2010<jats:styled-content style=\"fixed-case\">KO<\/jats:styled-content> than in <jats:styled-content style=\"fixed-case\">WT<\/jats:styled-content> mice. A receiver operating characteristic (<jats:styled-content style=\"fixed-case\">ROC<\/jats:styled-content>) analysis discriminated Cdkl5\u2010<jats:styled-content style=\"fixed-case\">KO<\/jats:styled-content> significantly from <jats:styled-content style=\"fixed-case\">WT<\/jats:styled-content> mice based on sleep apnea occurrence. These data demonstrate that sleep apneas are a core feature of <jats:styled-content style=\"fixed-case\">CDKL<\/jats:styled-content>5 disorder and a respiratory biomarker of <jats:styled-content style=\"fixed-case\">CDKL<\/jats:styled-content>5 deficiency in mice, and suggest that sleep\u2010disordered breathing should be evaluated routinely in <jats:styled-content style=\"fixed-case\">CDKL<\/jats:styled-content>5 patients.<\/jats:p>","DOI":"10.1111\/jsr.12512","type":"journal-article","created":{"date-parts":[[2017,2,23]],"date-time":"2017-02-23T10:22:42Z","timestamp":1487845362000},"page":"495-497","source":"Crossref","is-referenced-by-count":39,"title":["<scp>CDKL<\/scp>5 deficiency entails sleep apneas in mice"],"prefix":"10.1111","volume":"26","author":[{"given":"Viviana","family":"Lo Martire","sequence":"first","affiliation":[{"name":"Department of Biomedical and Neuromotor Sciences University of Bologna Bologna 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Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2017,2,23]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0091613"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/srep41698"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10072-009-0108-9"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/ejhg.2012.156"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.2012.04432.x"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1086\/375538"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-016-0418-y"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddi198"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0032-1308856"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.2951-12.2012"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2869.1995.tb00216.x"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0100536"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1113\/jphysiol.2006.119966"}],"container-title":["Journal 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